<p>Delandistrogene moxeparvovec was recently approved for Duchenne Muscular Dystrophy (DMD), using an adeno-associated virus to introduce a microdystrophin gene; however, the evidence is limited. The aim was to assess the effects of delandistrogene moxeparvovec on motor function and safety in the population with DMD. A systematic search of MEDLINE, Scopus, Web of Science and the Cochrane Library was conducted from their inception to May 2025. Clinical trials that assessed the effect or safety of delandistrogene moxeparvovec in the DMD population were included. For efficacy, the North Star Ambulatory Assessment (NSAA), Supine to Stand (TTSTAND), Climb 4 Stairs (TTCLIMB), 100-meter Timed Walk Test, and 10-meter Run Test were used, and for safety, the proportion of main adverse events was determined. Meta-analyses were performed for each outcome. Five trials involving 190 participants (4.8 to 6.0 years) were included. At one year, in the pre-post designs, there was an improvement of 2.63 points (95%CI: 1.74, 3.52) on the NSAA and − 0.29&#xa0;s (95%CI: -0.52, -0.06) on the TTSTAND, while compared with the placebo, there was an improvement of -0.64&#xa0;s (95%CI: -0.99, -0.30) in the TTSTAND. Moreover, the effect on some outcomes tended to be greater in participants aged 4–5 years. Severe side effects occurred in 10–15% of the samples. At one year after treatment, delandistrogene moxeparvovec had a modest effect on the progression of DMD. It is expected that future studies will confirm this efficacy, particularly in longer trials, as it is difficult to recover lost or ungained motor function.</p>